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Combining developmental and sleep health measures for autism spectrum disorder screening: an ECHO study

Pediatric Research (2024)Cite this article

Abstract

Background

Sleep problems are reported for up to 80% of autistic individuals. We examined whether parsimonious sets of items derived from the Modified Checklist for Autism in Toddlers, Revised (M-CHAT-R) and the Brief Infant Sleep Questionnaire (BISQ) are superior to the standard M-CHAT-R in predicting subsequent autism spectrum disorder (ASD) diagnoses.

Methods

Participants from 11 Environmental influences on Child Health Outcomes (ECHO) cohorts were included. We performed logistic LASSO regression models with 10-fold cross-validation to identify whether a combination of items derived from the M-CHAT-R and BISQ are superior to the standard M-CHAT-R in predicting ASD diagnoses.

Results

The final sample comprised 1552 children. The standard M-CHAT-R had a sensitivity of 44% (95% CI: 34, 55), specificity of 92% (95% CI: 91, 94), and AUROC of 0.726 (95% CI: 0.663, 0.790). A higher proportion of children with ASD had difficulty falling asleep or resisted bedtime during infancy/toddlerhood. However, LASSO models revealed parental reports of sleep problems did not improve the accuracy of the M-CHAT-R in predicting ASD diagnosis.

Conclusion

While children with ASD had higher rates of sleep problems during infancy/toddlerhood, there was no improvement in ASD developmental screening through the incorporation of parent-report sleep metrics.

Impact

  • Parental-reported sleep problems are common in autism spectrum disorder (ASD).

  • We investigated whether the inclusion of parental-reports of infant/toddler sleep patterns enhanced the effectiveness of developmental screening for autism.

  • We reported higher rates of difficulty falling asleep and resisting bedtime during infancy and toddlerhood among children later diagnosed with ASD; however, we did not find an improvement in ASD developmental screening through the incorporation of parent-report sleep metrics.

  • In our sample, the standard M-CHAT-R had a sensitivity of 39% among children of mothers with government insurance compared with a sensitivity of 53% among children of mothers with employer-based insurance.

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Fig. 1
Fig. 2
Fig. 3: Primary Model Performance.

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Data availability

Select de-identified data from the ECHO Program are available through NICHD’s Data and Specimen Hub (DASH). Information on study data not available on DASH, such as some Indigenous datasets, can be found on the ECHO study DASH webpage.

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Acknowledgements

The authors wish to thank our ECHO Colleagues; the medical, nursing, and program staff; and the children and families participating in the ECHO cohort.

Funding

Research reported in this publication was supported by the Environmental influences on Child. Health Outcomes (ECHO) Program, Office of the Director, National Institutes of Health, under. Award Numbers U2COD023375 (Coordinating Center), U24OD023382 (Data Analysis Center), U24OD023319 with co-funding from the Office of Behavioral and Social Science Research (PRO Core), UH3OD023251 (A.A.), UH3OD023320 (J.A.), UH3OD023313 (K.M.), UH3OD023318 (A.D.), UH3OD023279 (A.M.E.), UH3OD023271 (Karr), UH3OD023347 (Lester), UH3OD023349 (O.C.), UH3OD023348 (M.T.O.S.), and UH3OD023290 (Herbstman). The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health. The sponsor, NIH, participated in the overall design and implementation of the ECHO Program, which was funded as a cooperative agreement between NIH and grant awardees. The sponsor approved the Steering Committee-developed ECHO protocol and its amendments including COVID-19 measures. The sponsor had no access to the central database, which was housed at the ECHO Data Analysis Center. Data management and site monitoring were performed by the ECHO Data Analysis Center and Coordinating Center. All analyses for scientific publication were performed by the study statistician, independently of the sponsor. The lead author wrote all drafts of the manuscript and made revisions based on co-authors and the ECHO Publication Committee (a subcommittee of the ECHO Operations Committee) feedback without input from the sponsor. The study sponsor did not review or approve the manuscript for submission to the journal.

Author information

Author notes

  1. A list of members and their affiliations appears in the Supplementary Information.

Authors and Affiliations

  1. Department of Child and Adolescent Psychiatry, NYU Grossman School of Medicine, New York, NY, USA

    Lauren C. Shuffrey, Anahid Akbaryan, Julianna Collazo Vargas & Lauren Costello

  2. Department of Pediatrics, University of New Mexico, Albuquerque, NM, USA

    Brandon Rennie, Bennett L. Leventhal & Johnnye Lewis

  3. Department of Epidemiology, Johns Hopkins University Bloomberg School of Public Health, Baltimore, MD, USA

    Xiuhong Li, Noya Galai & Kristen L. McArthur

  4. Department of Statistics, University of Haifa, Mt Carmel, Israel

    Noya Galai

  5. Department of Psychiatry, Columbia University Irving Medical Center, New York, NY, USA

    Nicolò Pini, William P. Fifer, Maristella Lucchini, Ayesha Sania & Amy E. Margolis

  6. College of Engineering, Northeastern University, Boston, MA, USA

    Akram Alshawabkeh

  7. Department of Pediatrics, Hackensack Meridian School of Medicine, Nutley, NJ, USA

    Judy Aschner

  8. Department of Pediatrics, Brown University, Providence, RI, USA

    Viren D’Sa

  9. Maternal Newborn and Child Health: Discovery & Tools, The Bill & Melinda Gates Foundation, Seattle, WA, USA

    Sean Deoni

  10. Department of Gynecology & Obstetrics, Emory University, Atlanta, Georgia

    Anne Dunlop

  11. Center for Pediatric and Community Research, Avera Research Institute, Sioux Falls, SD, USA

    Amy J. Elliott

  12. Division of Developmental Neuroscience, New York State Psychiatric Institute, New York, NY, USA

    William P. Fifer

  13. Department of Child, Family, and Population Health Nursing, University of Washington, Seattle, WA, USA

    Jonica Hash

  14. Departments of Pediatrics and Psychiatry and Human Behavior, The Warren Alpert Medical School of Brown University, Providence, RI, USA

    Daphne Koinis-Mitchell

  15. Department of Medical Social Sciences, Northwestern University Feinberg School of Medicine, Chicago, IL, USA

    Jin-Shei Lai

  16. Department of Psychiatry and Behavioral Neuroscience, The University of Chicago, Chicago, IL, USA

    Bennett L. Leventhal

  17. Departments of Psychology and Pediatrics, Developmental and Brain and Cognitive Science Areas, University of Southern California, Los Angeles, CA, USA

    Santiago Morales

  18. Community Environmental Health Program, University of New Mexico Health Sciences Center, Albuquerque, NM, USA

    Sara S. Nozadi

  19. Department of Psychiatry, University of Rochester Medical Center, Rochester, NY, USA

    Thomas G. O’Connor

  20. Department of Pediatrics, University of North Carolina School of Medicine, Chapel Hill, NC, USA

    T. Michael O’Shea

  21. Analytics Program, RTI International, Research Triangle Park, NC, USA

    Grier P. Page

  22. School of Nursing, University of North Carolina, Chapel Hill, NC, USA

    Cathi Propper

  23. The Brown Center for the Study of Children at Risk, Woman & Infants Hospital of Rhode Island, Providence, RI, USA

    Coral Shuster

  24. Department of Communication Sciences and Disorders, Northeastern University, Chicago, IL, USA

    Emily Zimmerman

Authors
  1. Lauren C. Shuffrey
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  2. Brandon Rennie
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  3. Xiuhong Li
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  4. Noya Galai
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  5. Nicolò Pini
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  6. Anahid Akbaryan
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  7. Akram Alshawabkeh
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  8. Judy Aschner
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  9. Julianna Collazo Vargas
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  10. Lauren Costello
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  11. Viren D’Sa
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  12. Sean Deoni
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  13. Anne Dunlop
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  14. Amy J. Elliott
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  15. William P. Fifer
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  16. Jonica Hash
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  18. Jin-Shei Lai
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  19. Bennett L. Leventhal
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  20. Johnnye Lewis
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  21. Maristella Lucchini
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  23. Santiago Morales
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  24. Sara S. Nozadi
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  25. Thomas G. O’Connor
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  26. T. Michael O’Shea
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  27. Grier P. Page
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  28. Cathi Propper
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  29. Ayesha Sania
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  30. Coral Shuster
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  31. Emily Zimmerman
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  32. Amy E. Margolis
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Consortia

for the ECHO Cohort Consortium

Contributions

Conception and Design: Lauren C. Shuffrey, Brandon Rennie, Xiuhong Li, Noya Galai, Amy J. Elliott, William P. Fifer, and Amy E. Margolis. Acquisition of Data: Akram Alshawabkeh, Viren D’Sa, Sean Deoni, Judy Aschner, Anne Dunlop, Amy J. Elliott, Daphne Koinis-Mitchell, Coral Shuster, Thomas G. O’Connor, Michael T. O’Shea, and Cathi Propper. Analysis and Interpretation: Xiuhong Li, Noya Galai, Lauren C. Shuffrey, Brandon Rennie, Nicolò Pini, and Amy E. Margolis. All authors contributed to drafting and revising the article for intellectual content. All authors approve the final version of the manuscript.

Corresponding author

Correspondence to Lauren C. Shuffrey.

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Competing interests

The authors declare no competing interests.

Consent to participate

The study protocol was approved by the single ECHO institutional review board, WCG IRB. Written informed consent or parent’s/guardian’s permission was obtained along with child assent as appropriate, for the ECHO Cohort Data and Biospecimen Collection Protocol participation and for participation in specific study sites.

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Shuffrey, L.C., Rennie, B., Li, X. et al. Combining developmental and sleep health measures for autism spectrum disorder screening: an ECHO study. Pediatr Res (2024). https://doi.org/10.1038/s41390-024-03306-0

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